Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 16, 2021; 9(20): 5701-5708
Published online Jul 16, 2021. doi: 10.12998/wjcc.v9.i20.5701
Role of ranulas in early diagnosis of Sjögren’s syndrome: A case report
Na Chen, Da-Shun Zeng, Yu-Tong Su
Na Chen, Department of Endocrine and Metabolic Diseases, Shanghai Institute of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
Na Chen, Shanghai National Clinical Research Center for metabolic Diseases, Key Laboratory for Endocrine and Metabolic Diseases of the National Health Commission of the PR China, Shanghai National Center for Translational Medicine, Shanghai, China
Da-Shun Zeng, Department of Oral Surgery, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou 325200, Zhejiang Province, China
Yu-Tong Su, Department of Rheumatology and Immunology, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200025, China
Author contributions: Chen N and Su YT managed the case, and prepared and revised the manuscript; Zeng DS assisted with the preparation and revision of the manuscript; all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work; all authors take full responsibility for the integrity of the study and the final manuscript.
Supported by the National Natural Science Foundation of China, No. 81801600.
Informed consent statement: Informed consent was obtained from the patient for her inclusion in this report.
Conflict-of-interest statement: The authors declare no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Yu-Tong Su, MD, PhD, Doctor, Department of Rheumatology and Immunology, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, No. 197 Ruijin Second Road, Shanghai 200025, China. suyt2015@163.com
Received: March 9, 2021
Peer-review started: March 9, 2021
First decision: April 4, 2021
Revised: April 13, 2021
Accepted: May 24, 2021
Article in press: May 24, 2021
Published online: July 16, 2021
Abstract
BACKGROUND

Although the presentations of Sjögren’s syndrome (SS) are variable, ranging from mild dryness to wider systemic involvement, ranulas as early clinical signs were scarcely reported. Here, we present an adult patient with SS, who developed a unilateral simple ranula and was diagnosed primary SS 3 years later. We also provide a review of cases of SS and ranulas from 1980 to 2020.

CASE SUMMARY

A 22-year-old girl was found to have a left painless floor-of-mouth lesion 3 years ago, without obvious trauma or inducement. The diagnosis of a unilateral (left) simple ranula was made, and the ranula was surgically treated. Within 3 years after the ranula surgery, she developed acute lymphadenectasis in unilateral parotid twice without inducement, and ultrasonic examination revealed diffuse lesions in bilateral parotids and submandibular glands, which strongly suggested SS. Serologic tests and the unstimulated whole saliva flow rate confirmed the SS diagnosis.

CONCLUSION

Our study underlines that ranulas are early clinical signs of SS. As early diagnosis and early intervention of SS are important to obtain better outcomes, our findings underline the need for histopathological test after sublingual adenectomy and imaging detection of exocrine glands for the patients with ranulas.

Keywords: Sjögren’s syndrome, Ranulas, Early diagnosis, Parotitis, Case report

Core Tip: Although the presentations of Sjögren’s syndrome (SS) are variable, ranulas as early clinical signs were scarcely reported. Here, we present an adult patient with SS, who developed a unilateral simple ranula and was diagnosed with primary SS 3 years later. We also provide a review of cases of SS and ranulas from 1980 to 2020. By analyzing the symptoms, treatment, and prognosis of these patients, we propose that ranulas could be early clinical signs and manifestations of SS, which may raise the awareness of clinicians and lead to early interventions for SS in order to obtain better outcomes.